Cysticercosis is a very common disease in tropical countries like India. Cysticerci can be found anywhere in the body, but are most commonly detected in the brain, cerebrospinal fluid (CSF), skeletal muscle, subcutaneous tissue, or eye. Neurologic manifestations are the most common clinical feature. Isolated extra-central nervous system (CNS) involvement in cysticercosis is extremely rare. Herein, we have presented an interesting case of a young boy with a combination of hepatic and muscular cysticercosis without any evidence of CNS involvement.

Cysticercosis is caused by Cysticercus cellulosae, the larval form of the tape worm Taenia solium. Humans are the definitive host and the adult tapeworms reside in the small intestine. The larval forms are found in the skeletal muscles of the pig, the intermediate host, as well. To develop cysticercosis, a human has to replace the pig in T. solium life cycle and the eggs must mature within the human small intestine as they would do in the pig's intestine. Entry of the eggs into the human small intestine may occur through autoinfection or by ingestion or inhalation of egg-contaminated food or water. Finally, these cysticerci spreads through the intestinal wall and are carried by the blood stream to muscles, brain, and subcutaneous tissues, leading to clinical manifestations. [1]

Among the unusual form of clinical manifestation, there are very few case reports of isolated muscular cysticercosis and, to the best of our knowledge, hepatic cysticercosis has been reported only thrice before in medical literature. [2],[3],[4],[5],[6],[7],[8],[9]

Case Report

A 14-year-old boy was having intermittent type of fever in the range of 102-103° F along with headache for 3 weeks. Within 1 week of development of fever, he developed multiple superficial and deep swellings over the front of the chest, trunk, buttocks, and thigh. The superficial swellings were painful, erythematous, and papular. The deep swellings were present over right iliac fossa, buttocks, and thigh; they were soft, cystic, some were painful, and some painless, the largest of which measured approximately 3 x 2 cm. On systemic examination, a 3-cm, firm, non-tender liver was found to be palpable with a liver span of 16 cm.

Contrast enhanced computed tomography (CECT) of whole abdomen revealed multiple hypodense lesions in the right lobe of the liver (the largest one being 47 × 39 mm), both psoas muscles, erector spinae, and gluteus maximus, medius, and minimus on both sides, suggestive of parasitic cyst with a possibility of cysticercosis [Figure 1], [Figure 2], [Figure 3] and [Figure 4]. Ultrasound screening showed scolex-like appearance in the liver and intramuscular cysts, strongly favoring the possibility of cysticercosis [Figure 5] and [Figure 6]. On biopsy from the cysts within the muscles, there was structure of parasite, which included a prominent investing tegument or cuticle, aggregate of subcuticular cells, smooth muscle cells, and presence of sucker [Figure 7]. CT and magnetic resonance imaging (MRI) of brain were unremarkable and no cyst was found on fundoscopic examination. The antibody against Cysticercus by enzyme-linked immunosorbent assay (ELISA) was negative.

Figure 7: Biopsy taken from cyst within the muscles, there was structure of parasite that included a prominent investing tegument or cuticle, aggregate of subcuticular cells, smooth muscle cells and presence of sucker

After ruling out the presence of cysticerci in the fundus, he was put on steroids, followed by initiation of albendazole with slow up-titration to 400 mg, twice daily. As there are no definite guidelines available for dosage schedule of albendazole in such a scenario, we treated him in the same way as for neurocysticercosis. The patient started showing clinical improvement within 1 week and the swellings slowly reduced in size. He was discharged from the hospital after 2 weeks in a stable condition. Presently, on follow-up after another 2 weeks, he was doing well.

Discussion

Cysticercosis is an infection with the larval (Cysticercus) stage of T. solium. It is very common in a tropical country like India. Isolated muscular involvement is not very common and it usually occurs with neurologic involvement. Our patient had hepatic and muscular involvement without any neurological involvement along with the involvement of unusual sites like psoas muscles, gluteus maximus, medius, minimus, and erector spinae. Medical literature has only three previous case reports of hepatic cysticercosis, in a 62-year-old male having hepatic cysticercosis with history of colonic adenocarcinoma 9 years back, a 9-year-old girl with hepatic cysticercosis, and a 25-year-old male with hepatic cysticercosis, which was reported from India. [7],[8],[9] The absence of any neurological involvement (normal fundoscopy and normal brain imaging) makes this case a very atypical one. Although the antibody against Cysticercus came negative, the diagnosis of cysticercosis was confirmed beyond doubt by the ultrasound appearance of scolex and, subsequently, by tissue biopsy.

Conclusion

This case has been reported to show that truly extra-central nervous system (CNS) cysticercosis involving the liver and muscles does exist and due consideration needs to be given to this entity to formulate a definite treatment protocol. Unless more cases are reported and a standard guideline is published, such cases can be treated in the same manner as for neurocysticercosis.